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Lhx1/5 control dendritogenesis and spine morphogenesis of Purkinje cells via regulation of Espin.


ABSTRACT: In the cerebellar cortex, Purkinje cells (PCs) receive signals from different inputs through their extensively branched dendrites and serve as an integration centre. Defects in the dendritic development of PCs thus disrupt cerebellar circuitry and cause ataxia. Here we report that specific inactivation of both Lhx1 and Lhx5 in postnatal PCs results in ataxic mutant mice with abnormal dendritic development. The PCs in the mutants have reduced expression of Espin, an F-actin cytoskeleton regulator. We show that Espin expression is transcriptionally activated by Lhx1/5. Downregulation of Espin leads to F-actin mislocalization, thereby impairing dendritogenesis and dendritic spine maturation in the PCs. The mutant PCs therefore fail to form proper synapses and show aberrant electrophysiological properties. By overexpressing Espin, we can successfully rescue the defects in the mutant PCs. Our findings suggest that Lhx1/5, through regulating Espin expression, control dendritogenesis and spine morphogenesis in postnatal PCs.

SUBMITTER: Lui NC 

PROVIDER: S-EPMC5454373 | biostudies-literature | 2017 May

REPOSITORIES: biostudies-literature

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Lhx1/5 control dendritogenesis and spine morphogenesis of Purkinje cells via regulation of Espin.

Lui Nga Chu NC   Tam Wing Yip WY   Gao Caiji C   Huang Jian-Dong JD   Wang Chi Chiu CC   Jiang Liwen L   Yung Wing Ho WH   Kwan Kin Ming KM  

Nature communications 20170518


In the cerebellar cortex, Purkinje cells (PCs) receive signals from different inputs through their extensively branched dendrites and serve as an integration centre. Defects in the dendritic development of PCs thus disrupt cerebellar circuitry and cause ataxia. Here we report that specific inactivation of both Lhx1 and Lhx5 in postnatal PCs results in ataxic mutant mice with abnormal dendritic development. The PCs in the mutants have reduced expression of Espin, an F-actin cytoskeleton regulator  ...[more]

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